Abstract
Fungal sinusitis are very often caused by Aspergillus spp. Dematiaceae spp. and mucomycoses. Sinusitis with Scedosporium spp is very rare, especially in immunocompetent humans. A 42-year-old female presented with long history of facial pain and headache. Isolated opacification of maxillary sinus with double density sign was noted on left side in the CT scan. Diagnosed as fungal rhino sinusitis and endoscopic sinus surgery was carried out to clear the disease of the sinus and further mycological evaluation proved the organism to be Scedosporium apiospermum. We report the rare case of a non-invasive isolated fungal sinusitis caused by fungi Scedosporium apiospermum.
Supplementary Information
The online version contains supplementary material available at 10.1007/s12070-024-05167-9.
Keywords: Fungal rhino sinusitis, Mycology, Rhinology, Scedosporium
Background
Fungal sinusitis is classically divided into two groups; Non-invasive and Invasive. Invasive fungal sinusitis is again classified into Acute fulminant, Granulomatous and Chronic forms by De Shazo et al.[1] The pathogens mostly isolated in these cases are by Aspergillus spp. Dematiaceae spp. and mucomycoses. Scedosporium apiospermum is a rare pathogen seen in soil and polluted water. Although they have been identified to be associated with significant infections in immunocompromised individuals, non-invasive sinus infections in immunocompetent hosts are rarely reported.[2]
We report the rare case of a non-invasive isolated fungal sinusitis caused by fungi Scedosporium apiospermum in an otherwise healthy and immunocompetent individual.
Case Report
A 42-year-old homemaker lady visited the outpatient clinic with complaints of long history of recurrent headache and facial pain, mainly over left side of face. She also had occasional foul-smelling nasal discharge. Many courses of oral antibiotics and other topical medications were used in the past which showed little improvement. She did not have any other comorbid conditions in the past. Nasal endoscopic evaluation using a 0 degree Hopkins rod showed congestion of mucosa with minimal amount of mucopus draining from left middle meatus with obliteration of left osteomeatal area. A non-contrast computed tomography imaging of the nose and paranasal sinuses showed an isolated opacification of the left maxillary sinus with double dense shadows of the sinus contents and complete blockade of the left osteomeatal complex (Fig. 1). Other sinuses were free of disease. A diagnosis of fungal sinusitis was made.
Fig. 1.
Non contrast CT scan of paranasal sinuses showing isolated opacification of the left maxillary sinus with double dense shadows
Patient was taken up for functional endoscopic sinus surgery under general anaesthesia. Dirty white cheesy material was found filling the left maxillary sinus with congested inflamed mucosa of sinus (Fig. 2). Fungal materials were completely removed and diseased mucosa cleared followed by thorough sinus wash (Fig. 3). Post-operative period was uneventful and patient was relieved of all the symptoms. Follow-up nasal endoscopic evaluation showed clean sinuses.
Fig. 2.
Intra-operative endoscopic image of left maxillary sinus showing cheesy material filling the cavity
Fig. 3.
Intra-operative endoscopic image of left maxillary sinus after removal of fungal materials and sinus wash
Histopathological evaluation of the sinus mucosa showed inflammation without invasion of the mucous membrane by the fungal hyphae indicating a non-invasive fungal sinusitis. Mycology report came out as growth of Scedosporium apiospermum in the sample (Fig. 4). Patient was stable and review nasal endoscopy revealed clean sinus cavity and no further treatment was required.
Fig. 4.
Lactophenol cotton blue stain showing hyphae and budding yeast like structures
Discussion
Inflammatory changes of the nose and paranasal sinus mucosa due to fungal infections are the basic pathology of fungal sinusitis.[3] While acute fulminant invasive, chronic invasive and granulomatous fungal sinusitis are mostly seen among immunocompromised individuals, immunocompetent individuals develop non-invasive forms like allergic fungal sinusitis and sinus mycetoma. Patients with intractable sinusitis not responding to multiple antibiotic courses should be considered a candidate for fungal sinusitis.[1]
In this case report we report a non-invasive fungal sinusitis which is unique due to the organism caused this condition; Scedosporium apiospermum. This fungus is an inhabitant of soil and polluted water and is well recognised to cause mycetoma normally affecting extremities. It is also described to cause pulmonary fungomas.[4] Sinusitis caused by S. apiospermum is less commonly demonstrated and natural history of infection remains poorly identified. Literature shows cases of sinusitis caused by Pseudallescheria boydii whose imperfect form in Scedosporium apiospermum.[5] First case was reported by Gluckman et al. in 1977.[6]
Chronic non-invasive fungal sinusitis is usually seen in immunocompetent non-atopic individuals and is usually a nonprogressive disease that cause minimal symptoms. They tend to involve a single sinus, mostly the maxillary sinuses and cause minimal mucosal inflammation without bone erosion or mucosal invasion.[7] In our case also the disease was limited to left maxillary sinus only and CT scan images showed no evidence of any bony erosions. Histopathology evaluation described mucosal inflammation without invasion of fungal hyphae.
Differentiation of Scedosporium from Aspergillus is a challenging work for pathologists due to the morphological similarity in branching patterns. Aspergillus being the most common pathogen involved, there is always a tendency for erroneous reporting from pathologists.[3] Microbiological profiling using antifungals is a key factor to differentiate Scedosporium from Aspergillus Spp. Scedosporium spp. tends to resist many antifungals compared to Aspergillus.
Treatment protocol in non-invasive fungal sinusitis is usually straight forward. Complete removal of the fungal material from the sinus involved.[7] This patient was taken up for endoscopic sinus surgery eradication of the fungal materials from the sinus and ensure smooth aeration of the sinus cavity in the post-operative period.
Conclusion
Any patients with intractable sinusitis not responding to multiple antibiotic courses should prompt a chance of fungal sinusitis. Early intervention and thorough surgical removal of fungal matter will help complete recovery in non-invasive fungal sinusitis.
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Acknowledgements
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Author Contributions
All authors discussed the results and contributed to the final manuscript.
Funding
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Data Availability
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Competing Interests
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References
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Data Availability Statement
Yes, All data generated or analysed during this study are included in this published article.